Congenital Cutaneous Defects as Complications in Surviving Co-twins

Léauté‐Labrèze, Christine; Depaire-Duclos, Florence; Sarlangue, J.; Fontan, D; Sandler, Boris; Maleville, J; Taı̈eb, Alain

doi:10.1001/archderm.134.9.1121

Cited by 44 publications

(19 citation statements)

References 19 publications

(19 reference statements)

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“…Intra-uterine death of a twin may cause release of thrombotic material, promoting ACC in the surviving twin in the case of vascular shunts. This is usually associated with extensive placental infarctions, disseminated intravascular coagulation and other direct and usually severe injuries to the surviving twin, which we failed to fi nd in our index case (22,23) .…”

Section: Discussionmentioning

confidence: 63%

Aplasia cutis congenita in surviving co-twin after propylthiouracil exposure in utero

Löllgen

Calza

Schwitzgebel

et al. 2011

Journal of Pediatric Endocrinology and Metabolism

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Aim: Aplasia cutis congenita (ACC) has been observed after fetal exposure to the antithyroid drug methimazole (MMI), but not reported after propylthiouracil (PTU), the current antithyroid drug of choice during pregnancy. This occurrence has implications for patient information and causal research. Case report: We describe a surviving term co-twin to a mother with hyperthyroidism exposed to PTU from conception to 34 weeks of gestation presenting with ACC at birth. Discussion: The association between PTU exposure and ACC is clinically relevant and allows speculation on the etiology. A similar mechanism to the classical MMI-induced ACC is postulated, unless a vascular etiology suggested by a vanishing twin or maternal hyperthyroidism itself is causal. Coincidence of PTU exposure and ACC seems unlikely. Conclusion: ACC in a newborn after PTU exposure during pregnancy hitherto observed only after MMI strongly encourages further reports of similar cases that may remain clinically underdiagnosed or unreported. Such confi rmation could have signifi cant implications for maternal treatment of hyperthyroidism, common in women of childbearing age.

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Section: Discussionmentioning

confidence: 63%

Aplasia cutis congenita in surviving co-twin after propylthiouracil exposure in utero

Löllgen

Calza

Schwitzgebel

et al. 2011

Journal of Pediatric Endocrinology and Metabolism

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“…The first patient of Kline and Moore 4 had a pulseless left hand but a Doppler study was not mentioned. All other CVIC cases had detectable pulses 2–6 . Hence, the degree of ischaemia in neonatal gangrene is more severe and the affected extremity is more involved and is often not salvageable, in contrast to CVIC.…”

Section: Discussionmentioning

confidence: 96%

“…The syndrome is characterized by muscle necrosis and nerve palsy secondary to increased intracompartmental pressure 1 . However, congenital Volkmann ischaemic contracture (CVIC) is rare, and to our knowledge only 11 other cases have been reported, mainly in the literatures of plastic and orthopaedic surgery 2–7 . CVIC presents with various cutaneous manifestations including an initial blue plaque, pressure necrosis, bullae, and a healed eschar over the affected extremity (Table 1).…”

Section: Summary Of 11 Other Cases Of Congenital Volkmann Ischaemic Cmentioning

confidence: 99%

Congenital Volkmann ischaemic contracture: a case report and review

et al. 2004

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Congenital Volkmann ischaemic contracture or neonatal compartment syndrome has rarely been discussed in the literature of dermatology. The condition often involves the upper extremity with cutaneous lesions, contractures and neuropathy. Because the lesions can be mistaken for other entities including necrotizing fasciitis, neonatal gangrene, congenital varicella, aplasia cutis congenita, amniotic band syndrome, subcutaneous fat necrosis and epidermolysis bullosa, dermatologists play a significant role in the diagnosis and, consequently, the treatment of the patient. We describe a premature newborn who had a unilateral, well-demarcated necrotic plaque with a central pallor at birth. The plaque extended circumferentially over the left forearm from the wrist to the elbow. Left wrist oedema, bullae over the fingers and flaccid paralysis at the wrist were also noted.

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“…Type V consists of a symmetrical defect of the trunk and/or limbs in association with a fetus papyraceus, 1 which is more likely to be related to variation in blood flow through the placental blood vessels, embolic phenomena in vessels or a coagulopathy related to the dead fetus in utero. 2 The baby has remained well but the skin defect has been slow to epithelialise. Treatment is currently conservative with regular dressings and there remains an option to skin graft.…”

Section: Aplasia Cutis In Association With a Triplet Pregnancy And Fementioning

confidence: 99%